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Primary cilia

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https://www.readbyqxmd.com/read/28534617/comparative-proteomics-reveals-timely-transport-into-cilia-of-regulators-or-effectors-as-a-mechanism-underlying-ciliary-disassembly
#1
Limei Wang, Lixiao Gu, Dan Meng, Qiong Wu, Haiteng Deng, Junmin Pan
Primary cilia are assembled and disassembled during cell cycle progression. During ciliary disassembly, ciliary axonemal microtubules (MTs) are depolymerized accompanied with extensive posttranslational protein modifications of ciliary proteins including protein phosphorylation, methylation and ubiquitination. These events are hypothesized to involve transport of effectors or regulators into cilia at the time of ciliary disassembly from the cell body. To prove this hypothesis and identify new proteins involved in ciliary disassembly, we analyzed disassembling flagella in Chlamydomonas using comparative proteomics with TMT labeling...
May 23, 2017: Journal of Proteome Research
https://www.readbyqxmd.com/read/28529994/rapamycin-increases-length-and-mechanosensory-function-of-primary-cilia-in-renal-epithelial-and-vascular-endothelial-cells
#2
Rinzhin T Sherpa, Kimberly F Atkinson, Viviana P Ferreira, Surya M Nauli
Primary cilia arebiophysically-sensitive organelles responsible for sensing fluid-flow and transducing this stimulus into intracellular responses. Previous studies have shown that the primary cilia mediate flow-induced calcium influx, and sensitivity of cilia function to flow is correlated to cilia length. Cells with abnormal cilia length or function can lead to a host of diseases that are collectively termed as ciliopathies. Rapamycin, a potent inhibitor of mTOR (mammalian target of rapamycin), has been demonstrated to be a potential pharmacological agent against the aberrant mTOR signaling seen in ciliopathies such as polycystic kidney disease (PKD) and tuberous sclerosis complex (TSC)...
December 2016: Int Educ Res J
https://www.readbyqxmd.com/read/28513435/a-bioactive-peptide-amidating-enzyme-is-required-for-ciliogenesis
#3
Dhivya Kumar, Daniela Strenkert, Ramila S Patel-King, Michael T Leonard, Sabeeha S Merchant, Richard E Mains, Stephen M King, Betty A Eipper
The pathways controlling cilium biogenesis in different cell types have not been fully elucidated. We recently identified peptidylglycine α-amidating monooxygenase (PAM), an enzyme required for generating amidated bioactive signaling peptides, in Chlamydomonas and mammalian cilia. Here, we show that PAM is required for the normal assembly of motile and primary cilia in Chlamydomonas, planaria and mice. Chlamydomonas PAM knockdown lines failed to assemble cilia beyond the transition zone, had abnormal Golgi architecture and altered levels of cilia assembly components...
May 17, 2017: ELife
https://www.readbyqxmd.com/read/28504201/cilia-the-sensory-antennae-in-the-eye
#4
REVIEW
Helen May-Simera, Kerstin Nagel-Wolfrum, Uwe Wolfrum
Cilia are hair-like projections found on almost all cells in the human body. Originally believed to function merely in motility, the function of solitary non-motile (primary) cilia was long overlooked. Recent research has demonstrated that primary cilia function as signalling hubs that sense environmental cues and are pivotal for organ development and function, tissue hoemoestasis, and maintenance of human health. Cilia share a common anatomy and their diverse functional features are achieved by evolutionarily conserved functional modules, organized into sub-compartments...
May 11, 2017: Progress in Retinal and Eye Research
https://www.readbyqxmd.com/read/28500304/microgravity-induces-inhibition-of-osteoblastic-differentiation-and-mineralization-through-abrogating-primary-cilia
#5
Wengui Shi, Yanfang Xie, Jinpeng He, Jian Zhou, Yuhai Gao, Wenjun Wei, Nan Ding, Huiping Ma, Cory J Xian, Keming Chen, Jufang Wang
It is well documented that microgravity in space environment leads to bone loss in astronauts. These physiological changes have also been validated by human and animal studies and modeled in cell-based analogs. However, the underlying mechanisms are elusive. In the current study, we identified a novel phenomenon that primary cilia (key sensors and functioning organelles) of rat calvarial osteoblasts (ROBs) gradually shrank and disappeared almost completely after exposure to simulated microgravity generated by a random positioning machine (RPM)...
May 12, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28495666/how-to-use-nasal-nitric-oxide-in-a-child-with-suspected-primary-ciliary-dyskinesia
#6
Kim Simpson, Malcolm Brodlie
Measuring nasal nitric oxide (nNO) is increasingly used as part of testing for primary ciliary dyskinesia (PCD). The diagnosis of PCD is often delayed until after bronchiectasis is established and auditory damage has occurred. It is important that all paediatricians are aware of clinical features that are suggestive of PCD that should prompt diagnostic testing. nNO levels are recognised to be low in people with PCD and results generated by static chemiluminescence analysers using velum closure technique in older children have good sensitivity and specificity...
May 11, 2017: Archives of Disease in Childhood. Education and Practice Edition
https://www.readbyqxmd.com/read/28495528/hepatic-ischemia-reperfusion-injury-disrupts-the-homeostasis-of-kidney-primary-cilia-via-oxidative-stress
#7
Sang Jun Han, Hee-Seong Jang, Sung Young Seu, Hee-Jung Cho, Yoon Jin Hwang, Jee In Kim, Kwon Moo Park
Acute kidney injury (AKI) is a major complication of hepatic surgeries. The primary cilium protrudes to the lumen of kidney tubules and plays an important role in renal functions. Disruption of primary cilia homeostasis is highly associated with human diseases including AKI. Here, we investigated whether transient hepatic ischemia induces length change and deciliation of kidney primary cilia, and if so, whether reactive oxygen species (ROS)/oxidative stress regulates those. HIR induced damages to the liver and kidney with increases in ROS/oxidative stress...
May 8, 2017: Biochimica et Biophysica Acta
https://www.readbyqxmd.com/read/28494707/histologic-and-ultrastructural-findings-in-dogs-with-chronic-respiratory-disease-suspected-of-ciliary-dyskinesia
#8
Ileana C Miranda, Jennifer L Granick, Anibal G Armién
Mucociliary clearance is a main defense mechanism of the respiratory tract, which can be inherently impaired in primary ciliary dyskinesia (PCD) or reversibly altered in secondary ciliary dyskinesia (SCD). Limited diagnostic test availability likely leads to misdiagnosis or underdiagnosis of PCD in animals. This study evaluated the light and transmission electron microscopy (TEM) changes in the respiratory mucosa of 15 dogs with chronic respiratory disease suspected of PCD. Necropsy was performed in 1 case and 2 dogs were used as negative controls...
January 1, 2017: Veterinary Pathology
https://www.readbyqxmd.com/read/28493804/hereditary-renal-cystic-disorders-imaging-of-the-kidneys-and-beyond
#9
Jonathan R Dillman, Andrew T Trout, Ethan A Smith, Alexander J Towbin
The purpose of this article is to review the hereditary renal cystic diseases that can manifest in children and adults, with specific attention to pathogenesis and imaging features. Various common and uncommon hereditary renal cystic diseases are reviewed in terms of their underlying etiology, including the involved genetic mutations and the affected proteins and cellular structures. Focus is placed on the morphologic findings in each condition and the features that distinguish one disorder from another. The two most common categories of hereditary renal cystic disease are (a) the ciliopathic disorders, which are related to mutations affecting the primary cilia (called "ciliopathies"), and (b) the phakomatoses...
May 2017: Radiographics: a Review Publication of the Radiological Society of North America, Inc
https://www.readbyqxmd.com/read/28487520/an-ovine-hepatorenal-fibrocystic-model-of-a-meckel-like-syndrome-associated-with-dysmorphic-primary-cilia-and-tmem67-mutations
#10
C Stayner, C A Poole, S R McGlashan, M Pilanthananond, R Brauning, D Markie, B Lett, L Slobbe, A Chae, A C Johnstone, C G Jensen, J C McEwan, K Dittmer, K Parker, A Wiles, W Blackburne, A Leichter, M Leask, A Pinnapureddy, M Jennings, J A Horsfield, R J Walker, M R Eccles
Meckel syndrome (MKS) is an inherited autosomal recessive hepatorenal fibrocystic syndrome, caused by mutations in TMEM67, characterized by occipital encephalocoele, renal cysts, hepatic fibrosis, and polydactyly. Here we describe an ovine model of MKS, with kidney and liver abnormalities, without polydactyly or occipital encephalocoele. Homozygous missense p.(Ile681Asn; Ile687Ser) mutations identified in ovine TMEM67 were pathogenic in zebrafish phenotype rescue assays. Meckelin protein was expressed in affected and unaffected kidney epithelial cells by immunoblotting, and in primary cilia of lamb kidney cyst epithelial cells by immunofluorescence...
May 9, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28487361/biochemical-characterization-of-purified-mammalian-arl13b-indicate-that-it-is-an-atypical-gtpase-and-arl3-guanine-nucleotide-exchange-factor-gef
#11
Anna A Ivanova, Tamara Caspary, Nicholas T Seyfried, Duc M Duong, Andrew B West, Zhiyong Liu, Richard A Kahn
Primary cilia play central roles in signaling during metazoan development. Several key regulators of ciliogenesis and ciliary signaling are mutated in humans, resulting in a number of ciliopathies, including Joubert Syndrome (JS). ARL13B is a ciliary GTPase with at least three missense mutations identified in JS patients. ARL13B is a member of the ARF family of regulatory GTPases, but is atypical in having a non-homologous, C-terminal domain of ~20 kDa and at least one key residue difference in the consensus GTP binding motifs...
May 9, 2017: Journal of Biological Chemistry
https://www.readbyqxmd.com/read/28481653/accuracy-of-nasal-nitric-oxide-measurement-as-a-diagnostic-test-for-primary-ciliary-dyskinesia-a-systematic-review-and-meta-analysis
#12
Adam J Shapiro, Maureen Josephson, Margaret Rosenfeld, Ozge Yilmaz, Stephanie D Davis, Deepika Polineni, Elena Guadagno, Margaret W Leigh, Valery Lavergne
BACKGROUND: Primary Ciliary Dyskinesia (PCD) is a rare disorder causing chronic oto-sino-pulmonary disease, generally diagnosed through evaluation of respiratory cilia ultrastructure and/or genetic testing. Nasal nitric oxide (nNO) measurement is a PCD screening test, as PCD patients have low nNO levels, but its value as a diagnostic test remains unknown. OBJECTIVE: Perform a systematic review assessing the utility of nNO measurement (index test) as a diagnostic tool compared to the reference standard of electron microscopy (EM) evaluation of ciliary defects and/or detection of biallelic mutations in PCD genes...
May 8, 2017: Annals of the American Thoracic Society
https://www.readbyqxmd.com/read/28479679/primary-cilia
#13
Deepak Venkatesh
No abstract text is available yet for this article.
January 2017: Journal of Oral and Maxillofacial Pathology: JOMFP
https://www.readbyqxmd.com/read/28474959/is-a-multidisciplinary-aerodigestive-clinic-more-effective-at-treating-recalcitrant-aerodigestive-complaints-than-a-single-specialist
#14
Janine M Rotsides, Gina M Krakovsky, Dinesh K Pillai, Sona Sehgal, Maura E Collins, Carolyn E Noelke, Nancy M Bauman
OBJECTIVE: To determine the utility of a pediatric multidisciplinary aerodigestive clinic (ADC) in treating recalcitrant aerodigestive conditions. METHODS: Longitudinal observational study of presenting complaints, evaluation, management, and outcome of patients seen during 12 monthly ADCs beginning August 2013. RESULTS: Fifty-five patients were seen by the ADC team (otolaryngology/gastroenterology/pulmonology/speech pathology/nurse practitioner) and followed for a mean 17...
May 1, 2017: Annals of Otology, Rhinology, and Laryngology
https://www.readbyqxmd.com/read/28473847/the-radial-organization-of-neuronal-primary-cilia-is-acutely-disrupted-by-seizure-and-ischemic-brain-injury
#15
Gregory W Kirschen, Hanxiao Liu, Tracy Lang, Xuelin Liang, Shaoyu Ge, Qiaojie Xiong
BACKGROUND: Neuronal primary cilia are sensory organelles that are critically involved in the proper growth, development, and function of the central nervous system (CNS). Recent work also suggests that they signal in the context of CNS injury, and that abnormal ciliary signaling may be implicated in neurological diseases. METHODS: We quantified the distribution of neuronal primary cilia alignment throughout the normal adult mouse brain by immunohistochemical staining for the primary cilia marker adenylyl cyclase III (ACIII) and measuring the angles of primary cilia with respect to global and local coordinate planes...
April 2017: Frontiers in Biology
https://www.readbyqxmd.com/read/28473325/a-novel-role-for-primary-cilia-in-airway-remodeling
#16
Carol S Trempus, Weifeng Song, Ahmed Lazrak, Zhihong Yu, Judy R Creighton, Bethany M Young, Rebecca L Heise, Yen R Yu, Jennifer L Ingram, Robert M Tighe, Sadis Matalon, Stavros Garantziotis
Primary cilia (PC) are solitary cellular organelles that play critical roles in development, homeostasis, and disease pathogenesis by modulating key signaling pathways such as sonic hedgehog and calcium flux. The antenna-like shape of PC enables them also to facilitate sensing of extracellular and mechanical stimuli into the cell, and a critical role for PC has been described for mesenchymal cells such as chondrocytes. However, nothing is known about the role of PC in airway smooth muscle cells (ASMC) in the context of airway remodeling...
May 4, 2017: American Journal of Physiology. Lung Cellular and Molecular Physiology
https://www.readbyqxmd.com/read/28468945/primary-cilia-control-the-maturation-of-tubular-lumen-in-renal-collecting-duct-epithelium
#17
Thomas Ernandez, Olga Komarynets, Alexandra Chassot, Soushma Sougoumarin, Priscilla Soulie, Yubao Wang, Roberto Montesano, Eric Feraille
The key role of the primary cilium in developmental processes is illustrated by ciliopathies resulting from genetic defects of its components. Ciliopathies include a large variety of dysmorphic syndromes that share in common the presence of multiple kidney cysts. These observations suggest that primary cilia may control morphogenetic processes in the developing kidney. In this study, we assessed the role of primary cilium in branching tubulogenesis and/or lumen development using kidney collecting duct-derived mCCDN21 cells that display spontaneous tubulogenic properties when grown in collagen/matrigel matrix...
May 3, 2017: American Journal of Physiology. Cell Physiology
https://www.readbyqxmd.com/read/28459465/intu-is-essential-for-oncogenic-hh-signaling-through-regulating-primary-cilia-formation-in-basal-cell-carcinoma
#18
N Yang, E L-H Leung, C Liu, L Li, T Eguether, X-J Jun Yao, E C Jones, D A Norris, A Liu, R A Clark, D R Roop, G J Pazour, K R Shroyer, J Chen
Inturned (INTU), a cilia and planar polarity effector, performs prominent ciliogenic functions during morphogenesis, such as in the skin. INTU is expressed in adult tissues but its role in tissue maintenance is unknown. Here, we report that the expression of the INTU gene is aberrantly elevated in human basal cell carcinoma (BCC), coinciding with increased primary cilia formation and activated hedgehog (Hh) signaling. Disrupting Intu in an oncogenic mutant Smo (SmoM2)-driven BCC mouse model prevented the formation of BCC through suppressing primary cilia formation and Hh signaling, suggesting that Intu performs a permissive role during BCC formation...
May 1, 2017: Oncogene
https://www.readbyqxmd.com/read/28456657/topography-of-calcium-phosphate-ceramics-regulates-primary-cilia-length-and-tgf-receptor-recruitment-associated-with-osteogenesis
#19
Jingwei Zhang, Melis Dalbay, Xiaoman Luo, Erik Vrij, Davide Barbieri, Lorenzo Moroni, Joost D de Bruijn, Clemens A van Blitterswijk, Paul Chapple, Martin M Knight, Huipin Yuan
The surface topography of synthetic biomaterials is known to play a role in material-driven osteogenesis. Recent studies show that TGFβ signaling also initiates osteogenic differentiation. TGFβ signaling requires the recruitment of TGFβ receptors (TGFβR) to the primary cilia. In this study, we hypothesize that the surface topography of calcium phosphate ceramics regulates stem cell morphology, primary cilia structure and TGFβR recruitment to the cilium associated with osteogenic differentiation. We developed a 2D system using two types of tricalcium phosphate (TCP) ceramic discs with identical chemistry...
April 26, 2017: Acta Biomaterialia
https://www.readbyqxmd.com/read/28453661/photoreceptor-outer-segment-as-a-sink-for-membrane-proteins-hypothesis-and-implications-in-retinal-ciliopathies
#20
Seongjin Seo, Poppy Datta
The photoreceptor outer segment (OS) is a unique modification of the primary cilium, specialized for light perception. Being homologous organelles, the primary cilium and the OS share common building blocks and molecular machinery to construct and maintain them. The OS, however, has several unique structural features that are not seen in primary cilia. Although these unique features of the OS have been well documented, their implications in protein localization have been under-appreciated. In this review, we compare the structural properties of the primary cilium and the OS, and propose a hypothesis that the OS can act as a sink for membrane proteins...
April 26, 2017: Human Molecular Genetics
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